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Review

A systematic review of
the effects of early
intervention on motor
development
Cornill H Blauw-Hospers MSc;
Mijna Hadders-Algra* MD PhD, Department of Neurology
Developmental Neurology, University of Groningen,
Groningen, the Netherlands.

*Correspondence to second author at University Hospital


Groningen, Developmental Neurology, Hanzeplein 1,
9713 GZ Groningen, the Netherlands.
E-mail: m.hadders-algra@med.rug.nl

We present a systematic review on the effect of early In the past few decades the importance of early intervention
intervention, starting between birth and a corrected age of 18 (EI) has become widely recognized. But what exactly is EI?
months, on motor development in infants at high risk for, or Typically, a single definition is used, which applies to EI for chil-
with, developmental motor disorders. Thirty-four studies dren at biological risk for developmental disorders and
fulfilled the selection criteria. Seventeen studies were performed children with developmental disabilities. Early Intervention
within the neonatal intensive care unit (NICU) environment. consists of multidisciplinary services provided to children from
Eight studies had a high methodological quality. They evaluated birth to 5 years of age to promote child health and well-being,
various forms of intervention. Results indicated that the enhance emerging competencies, minimize developmental
Newborn Individualized Developmental Care and Assessment delays, remediate existing or emerging disabilities, prevent
Program (NIDCAP) intervention might have a temporary functional deterioration, and promote adaptive parenting
positive effect on motor development. Twelve of the 17 post- and overall family functioning. These goals are accomplished
NICU studies had a high methodological quality. They addressed by individualized developmental, educational, and ther-
the effect of neurodevelopmental treatment (NDT) and specific apeutic services for children provided in conjunction with
or general developmental programmes. The results showed mutually planned support for their families. 1 In general, EI
that intervention in accordance with the principles of NDT does programmes use techniques derived from the domains of
not have a beneficial effect on motor development. They also physiotherapy, occupational therapy, developmental psychol-
indicated that specific or general developmental programmes ogy, and education. Little attention is paid to the effect of
can have a positive effect on motor outcome. We concluded that nutrition, even though it is well known that the cognitive
the type of intervention that might be beneficial for infants at outcome of breastfed children is significantly better than that
preterm age differs from the type that is effective in infants who of formula-fed children.2
have reached at least term age. Preterm infants seem to benefit The earliest studies on EI programmes primarily addressed
most from intervention that aims at mimicking the intrauterine improvement in motor skills. Later, the focus shifted towards
environment, such as NIDCAP intervention. After term age, family-focused and other functional outcomes.3 It seems,
intervention by means of specific or general developmental therefore, that EI serves as an umbrella term covering the
programmes has a positive effect on motor development. whole field of childhood intervention.
One of the problems associated with the use of the term EI
is the interpretation of early. Early can be understood in
two ways, namely as early in life and as early in the expres-
sion of the condition. Each of the two types of earliness is
associated with advantages and disadvantages for interven-
tion. The major advantage of intervening early in life is that the
brain is considered to be very plastic at this time. The brain is
especially plastic in the phase occurring after the completion
of neuronal migration during which the processes of dendritic
outgrowth and synapse formation are highly active.4 This
means that high plasticity can be expected between 2 to 3
months before, and about 6 to 8 months after term age.5
See end of paper for list of abbreviations. However, there are two potential disadvantages that might be

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associated with intervention early in life. First, the type of prob- participants were infants with high biological risk for, or with
lems that the infant will develop later in life will not yet be clear. developmental disabilities; (2) the aim of the intervention
This makes effective goal setting for EI difficult. Second, part of (mostly among others) was to improve motor development;
the at-risk population will not develop a developmental disor- (3) the onset of intervention in at least 50% of the participants
der, thereby making intervention, perhaps, superfluous for was between birth and the (corrected) age of 18 months; and
these children. (4) the journal in which the study was published had an impact
Intervention in children with a developmental disorder factor of more than 0.3. Excluded from the review were studies
generally starts later in life, i.e. at the time during infancy or restricted to medical and orthopaedic interventions and stud-
preschool age when the condition is expressed in dysfunc- ies in populations of healthy low-risk preterm infants or in
tion. Two advantages of the latter situation are that interven- populations of socially disadvantaged children without spe-
tion is applied to children who are really in need of EI and cific biological risk for developmental disorders. On the basis
that the goals of the intervention can be formulated relatively of the abstract, 60 papers were selected as potential candi-
easily. The most important disadvantage of intervention that dates for the review. After each entire paper had been read,
starts when the disorder has become undeniable is that it there remained 36 original studies that met all inclusion crite-
starts relatively late from the point of view of plasticity of the ria. There were two studies within this selection for which the
brain.5 Indeed, previous studies have indicated that pro- results were published in more than one paper.811 In the pre-
grammes starting before the ninth month after term provid- sent review these studies were treated as single entities. The
ed more improvement in both motor and personal skills papers not included in the review were mainly review articles,
than programmes starting later.6,7 In view of this, it seems studies focusing on improvement in cognitive function, and
wise to start intervention early in life. studies in which the intervention was applied after the age of
The aim of the present study was, therefore, to conduct a 18 months.
systematic review of studies on intervention starting early in
life in children at high biological risk for developmental disor- EVALUATION PROCEDURE
ders. In particular, we attempted to unravel the elements that The evaluation of the studies focused on the type and size of
might contribute to a beneficial effect on motor develop- groups included in the study, the level of evidence (Table I),
ment. Specific attention was paid to the timing of interven- and the internal and external validity of the study (criteria
tion, to see whether we could find indications for the existence specified in Table II). The level of evidence of a study and its
of a crucial age period during which EI results in the most ben- internal and external validity were the determinants for the
eficial outcome. methodological quality of the study. All studies were rated
independently by both reviewers. Interrater agreement was
Method calculated for each of the determinants of methodological
SELECTION PROCEDURE quality. Agree- ment was high: Cohens kappa varied from 0.86
A literature search was performed with the following electronic (internal validity), 0.94 (level of evidence) to 0.95 (external
databases: MEDLINE (1966 to July 2004), Cinahl (1982 to July validity). Disagreements were discussed until consensus was
2004), AMED (1985 to July 2004), PsycINFO (1967 to July reached. In addition, attention was paid to specifics of the
2004), and PEDro (1992 to 2004). Reference lists in original intervention programme, such as the type of intervention, its
studies and reviews were also examined for appropriate arti- period of application and intensity, the location where the
cles. The keyword early intervention initially revealed 13 699 intervention had been performed, and parental involvement
hits. To reduce the number of hits the following additional key- (Table II). Finally, age of infants at evaluation, outcome mea-
words were used: infant, motor development, low birth sures, and results were specified to evaluate further the effect
weight, preterm, high-risk, and cerebral palsy (CP). There- of the intervention.
after the number of hits was reduced to 485. To assess the effect of the age period during which EI
Studies from these 485 papers were included in the review took place, we divided the studies into three age groups.
only when they fulfilled the following four criteria: (1) the The first group consisted of studies dealing with intervention

Table I: Levels of evidencea

Level Group research Single individual research

I Randomized controlled trials n=1 randomized controlled trial


II Non-randomized controlled trials ABABA design
Prospective cohort studies with concurrent control group Alternating treatments (e.g. ABACA)
Multiple baseline across participants
III Case studies with control participants ABA design
Cohort studies with historical control group
IV Case series without control participants AB design
V Case reports Case reports
Non-empirical methods Non-empirical methods
aFrom Sackett (1989)12 and Butler and Darrah (2001).57

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programmes applied in the neonatal intensive care unit category of age at onset of intervention was added, consist-
(NICU); the second group contained studies in which the ing of studies in which intervention started at some age bet-
intervention programme started between discharge from ween discharge from the NICU and 18 months corrected age.
NICU care and a corrected age of 9 months; the third group Within the age periods, studies were ranked primarily accor-
included studies in which intervention started between a ding to the level of evidence and were next ranked by inter-
corrected age of 9 and 18 months. nal validity, external validity, and effect (Tables III to VI).
The studies were found to be so heterogeneous in the
type of intervention evaluated, the outcome measures used POPULATIONS AND METHODS OF INTERVENTION
to evaluate the effect of intervention, and the age at which The number of children included in the studies ranged from
outcome was studied that no meta-analysis to examine effect 10 to 746 individuals (median 44.5; Table III). Information
sizes of interventions could be performed. about the rate of attrition was provided in 33 studies: it var-
ied from 0% to 64%, with a median of 13%. In most studies
Results (24 of 34) the study group consisted of so-called high-risk
Seventeen of the 34 studies dealt with interventions performed infants, i.e. infants born preterm or with a low birthweight. The
within an NICU setting, eight evaluated intervention starting remaining studies evaluated the effect of intervention in
between discharge from the NICU and 9 months, and six studies infants with CP, delayed cognitive and motor development,
assessed intervention which started between 9 and 18 months or Down syndrome.
corrected age. In three studies the age at onset of interven- The intervention programmes applied showed consider-
tion exceeded the preset period criteria. Therefore, a fourth able diversity (Table IV). In 29 studies, at least brief information

Table II: Evaluation criteria (modification of Siebes et al.51)

Group and design Description

Sample size Total, experimental group, control or contrast group


? Unspecified
Attrition Number
? Unspecified
Study group High-risk (i.e. preterm/low birthweight), CP, Down syndrome
? Unspecified
Levels of evidence Levels according to Sacketts method for grading research12 modified by Butler and Darrah57; see Table I
Internal validity: ++ High internal validity
can measured effects + Fair internal validity
be attributed to Low internal validity
intervention under Internal validity can be reduced by various study variations such as participant assignment, contamination,
study? co-intervention, and blind assessment
External validity: can ++ Generalization is plausible
results of research + Some possibilities for generalization
be generalized? No information about generalization
Methodological quality Based on level of evidence and internal and external validity
Intervention
Method of treatment
Contents description ++ Detailed description
+ Summary only
Very limited information
Period of application Description
? Unspecified
Intensity Description
? Unspecified
Location Hospital, home, centre
Combination
Parental involvement P Enhance parental skills
T Parent is therapist
? Unspecified
Evaluation of effect
Age at evaluation Preterm, term, 19mo after term, 918mo, more than 18mo
Outcome measures Neuromotor
Developmental
Results E=C, E>C, E<C

CP, cerebral palsy; P, parent; T, therapist; E, experimental group; C, control group.

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about the intervention method was given; of these, 10 stud- that started after discharge from the NICU, neurodevelop-
ies gave a more detailed description. To obtain some insight mental treatment (NDT), which consists of a mix of general
into the intervention strategies and procedures applied, we sensory stimulation and passive and active motor interven-
assessed whether programmes contained the following ele- tion strategies, was the intervention most frequently used.
ments: procedures to reduce stress, sensory stimulation Other frequently applied forms of intervention were various
(specific unimodal, specific multimodal, general multimodal), developmental programmes, which always included general
motor intervention strategies (passive handling techniques, sensory stimulation and general stimulation of motor devel-
active training of specific motor abilities, general motor train- opment but could also imply passive handling techniques
ing), and parentinfant interaction strategies (Table V). Both and the enhancement of parentinfant interaction.
authors assessed the composition of the intervention pro- Information about the period of application of the interven-
grammes independently. Interrater agreement was high: tion was supplied in 30 studies. Most NICU interventions were
Cohens kappa for the various components varied from 0.79 applied during variable periods, because most interventions
to 1.00. NICU intervention programmes consisted mainly of took place between the age of some postnatal days until dis-
combinations of procedures aimed at reducing stress, the charge. The application period in the post-NICU studies varied
provision of auditory, tactile, visual or vestibular stimuli, and between 2 months and more than 4 years. In addition, the
passive motor handling procedures. Among the programmes intensity of intervention showed considerable heterogeneity.

Table III: Groups and design

Study n E C Attrition Study group Evidence Internal External


(%) level validity validity

NICU
Als et al.13 38 20 18 0 HR I ++ ++
Resnick et al.8,9 255 107 114 13 HR I + +
Darrah et al.14 107 53 54 51 HR I + +
Ariagno et al.15 35 14 14 20 HR I + +
Charpak et al.17 746 382 364 16 HR I + +
Nelson et al.16 37 21 16 30 HR or CNS injury I + +
Kleberg et al.18 25 11 9 20 HR I + +
Tessier et al.19 431 183 153 22 HR I + +
Westrup et al.28 41 21 20 37 HR I +
Korner et al.21 56 12 8 64 HR I
Feldman et al. 24 146 73 73 9 HR II
Helders et al.25 149 67 82 34 HR II
Als et al.22 16 8 8 0 HR III +
Leib et al.20 28 14 14 0 HR III
Mouradian and Als 23 40 20 20 0 HR III
Becker et al.26 38 ? ? 61 HR III
Kleberg et al.27 33 15 18 ? HR III
After NICU to 9mo
Goodman et al.29 80 40 40 01 HR I ++ ++
Piper et al.30 134 66 68 14 HR I ++ ++
Leksculchai and Cole 34 84 43 41 14 HR I ++ +
Barrera et al.33 80 32 48 26 HR I ++ +
Weindling et al.32 105 51 54 21 HR of CP I ++ +
Rothberg et al.31 49 28 21 39a HR I + +
dAvignon et al.35 32 12/10b 8 6 HR I +
Kanda et al.36 10 5 5 0 CP III
918mo
Ulrich et al.37 30 15 15 0 DS I ++ ++
Palmer et al.10,11 48 25 23 2 CP I ++ ++
Reddihough et al.38 66 32 34 9 CP I + +
Mahoney39 50 28 22 0 CP/ DS II ++ +
Eickmann et al.40 156 78 78 13 Delayed cogn/mot dev II + +
Piper and Pless58 37 21 16 0 DS III +
After NICU to 18mo
Mayo42 29 17 12 0 Suspected CP I ++ +
Scherzer et al.43 24 14 8 8 CP I ++ +
Harris 41 20 10 10 0 DS I +
aIn time between two studies, 31 children were lost to follow-up. bTen infants received a different kind of intervention. C, control/contrast

group; CNS, central nervous system; CP, cerebral palsy; Delayed cogn/mot dev, delayed cognitive/motor development; DS, Down syndrome;
E, experimental group; HR, high risk; ?, no information available; +, high; ++, fair; , low.

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It ranged from once a month to continuous intervention. home care. In 24 of the 34 studies, parents were incorporat-
Programmes starting after discharge from the NICU were ed into the intervention. In 19 studies the focus was on
mostly applied within a home-based setting. Another fre- enhancing parental skills, in the other five the parents per-
quently used method of supplying intervention was the use of formed (part of) the intervention, in other words, the par-
hospital-based or centre-based intervention combined with ents functioned as therapists.

Table IV: Type of intervention

Study Method Description Period of application Intensity Location Parents


Onset End intervention

NICU
Als et al.13 NIDCAP ++ 3d after birth ? Continuously Hp P
Resnick et al.8,9 Dev int ++ Birth 24mo Hp: daily Hp/Hm P
Hm: 2/mo
Darrah et al.14 Wb + 27d after birth Placement in Continuously Hp ?
open cots
Ariagno et al.15 NIDCAP + ? ? Continuously Hp P
Charpak et al.17 KC + 35d after birth Position no Continuously Hp P
longer accepted
Nelson et al.16 ATVV ++ 33wk PMA 2mo Hp: 15min, 2/d 5/wk Hp/Hm T
Hm: 2/d
Kleberg et al.18 NIDCAP + 1d after birth ? Continuously Hp P
Tessier et al.19 KC ++ Condition stable Discharge Continuously Hp P
Westrup et al.28 NIDCAP + 1d after birth 36wk PMA Continuously Hp P
Korner et al.21 Wb + <4d after birth Until evaluation Continuously Hp ?
Feldman et al. 24 KC ++ 3134wk PMA Discharge, >1h/d Hp P
duration 14d
Helders et al.25 T stim/RF Birth Discharge ? Hp ?
Als et al.22 NIDCAP ++ 9d after birth Discharge Continuously Hp P
Leib et al.20 Sens enr + Birth Discharge ? Hp ?
Mouradian and Als23 NIDCAP + ? <40wk PMA Continuously Hp P
Becker et al.26 Dev hand + Birth 36wk PMA ? Hp ?
Kleberg et al.27 NIDCAP + 3d after birth 36wk PMA ? Hp P
After NICU to 9mo
Goodman et al.29 NDT 3mo 12mo I: 1/mo >45min Hp/Hm P
P: daily (home
programme)
Piper et al.30 NDT + Term age 12mo I: 03mo: 1/wk Hm P
312mo: 1/2wk
P: daily
Leksculchai and Cole34 Dev pgm ++ Term age 4mo I: 1/mo Hm T
P: daily
Barrera et al.33 DPI/PIT + 4mo 16mo I: 47 mo: 12 h/wk Hm P
713 mo: 1/2wk
1316mo: 1/mo
Weindling et al.32 NDT Term age 12mo I: 06mo: 1/wk Hm ?
(CPcontinue) 69mo: 1/2wk
912mo: 1/mo
Rothberg et al.31 NDT + 3mo 12mo I: 1/mo >45min Hp/Hm P
P: daily (home
programme)
dAvignon et al.35 Vojta/NDT 47mo NDT: >3mo ? ? ?
Vojta: >6mo
Kanda et al.36 Vojta 1mo E: Mean duration E: 30min, 34/day Hm T
52mo
C: Mean duration
25mo

ATVV, auditorytactilevisualvestibular stimulation; C, control group; Ce, centre; CE, conductive education; Dev hand, developmental
handling; Dev int, developmental intervention; Dev mile, developmental milestones; Dev pgm, developmental programme; Dev S,
developmental skills; Dev stim, developmental stimulation; DPI, developmental programme intervention; E, experimental group;
Hm, home, Hp, hospital; I, instruction; IS, infant stimulation; KC, Kangaroo Care; mo, months (corrected age); NDT, neurodevelopmental
treatment; NIDCAP, Newborn Individualized Developmental Care and Assessment Program; P, parents; P, enhance parental skills;
PIT, parentinfant treatment; PMA, postmenstrual age; Phys T, physical therapy; Sens enr, sensory enrichment; T, parents are therapist;
T stim/RF tactile stimulation/range finding; TT, treadmill training; Wb, waterbed; ?, no information available. continued

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GENERAL EVALUATION OF EFFECTS (TABLES III TO VI ) the methodological strength of the studies.
Most studies evaluated the effect of intervention on motor
performance (1) during the intervention, (2) immediately EFFECT OF INTERVENTION STARTING IN THE NICU
after the end of intervention, and/or (3) some months or 1 to From the 17 NICU studies, eight had a high methodological
2 years after intervention. Various outcome measures were quality. These studies had an evidence level of I, a fair to high
used for effect evaluation. We categorized the measures as internal validity, and provided at least some possibilities for
either neuromotor, or tests that provide a more general generalization. In two of these eight studies a significantly
description of the childs developmental level (Table VII). positive effect of intervention on motor outcome could be
The Bayley Scales of Infant Development were most fre- demonstrated. One of the positive studies dealt with the effect
quently used as outcome measure for both neuromotor (Phys- of NIDCAP.13 The focus of intervention in this study was stress
ical Development Index; PDI) and developmental (Mental reduction in combination with general sensory stimulation.
Development Index; MDI) outcome. In 26 studies develop- The other intervention was aimed at improving the infants
mental tests were used to evaluate the effect of intervention. In general developmental level by means of a developmental pro-
nine of these, a beneficial effect of intervention on the develop- gramme including general sensory stimulation, general stimu-
mental parameters was reported. In 26 studies neuromotor lation of motor development, passive handling techniques,
tests were used. In 13 of these, study infants had a better neuro- and the enhancement of parentinfant interaction.8,9 In the
motor outcome than control infants. other six NICU studies of high methodological quality, inter-
Most studies were designed as randomized controlled vention had no statistically significant effect on motor develop-
trials. Twenty-three of the 34 studies had the highest level of ment.1419 The interventions used in these studies all included
evidence, namely level I, according to Sackett12 (see Table procedures to reduce the infants level of stress and multi-
I), four studies had a grade II level of evidence, and seven modal sensory stimulation (either specific or general), which
studies were classified as level III. Internal validity was high was or was not combined with passive motor intervention
in 11 studies, fair in 15, and low in eight. External validity techniques or the facilitation of parentinfant interaction.
was in general moderate only: in five studies generalization Five of the nine NICU studies with a lower methodological
was plausible, 17 studies offered some possibilities for gen- quality pointed to a positive effect of intervention.2024 Inter-
eralization, and 12 studies had low external validity. The vention used in these studies consisted of various combina-
validity of post-NICU studies was usually better than that of tions of procedures to reduce stress, multimodal sensory
NICU studies. stimulation, passive motor intervention strategies, or the
In the next sections we report the effects of intervention facilitation of parentinfant interaction. The remaining four
on motor development for the different age periods during studies were unable to demonstrate a beneficial effect of
which intervention had started, while taking into account intervention.2528

Table IV: continued

Study Method Description Period of application Intensity Location Parents


Onset End intervention

918mo
Ulrich et al.37 TT + 912mo Independent 8min/d, 5d/wk Hm T
walking
Palmer et al.10,11 NDT/IS ++ 1219mo Duration 12mo I: 1h 1/2wk Ce/Hm P
P: daily (home
programme)
Reddihough et al.38 CE ++ 1236mo Duration 6mo E: 2.8h/wk Ce ?
C: 2.9h/wk
Mahoney39 NDT/Dev S + Mean age 14mo Duration 12mo 3/mo, 45min Ce/Hm ?
Eickmann et al.40 Dev stim + 13mo 18mo I: 11 home visits Hm P
3045min,
3 workshops
Piper and Pless58 Dev mile + Mean age 9mo Duration 6mo I: 1h 1/2wk Ce/Hm P
P: home programme
After NICU to 18mo
Mayo42 NDT + 418mo Duration 6mo E: 1h/wk Hp/Hm P
C: 1h/mo
Scherzer et al.43 Phys T + 517mo 24mo 1h 1/2wk Hm T
Harris41 NDT ++ 221mo Duration 9wk E: 3/wk >40min Hm ?

ATVV, auditorytactilevisualvestibular stimulation; C, control group; Ce, centre; CE, conductive education; Dev hand, developmental
handling; Dev int, developmental intervention; Dev mile, developmental milestones; Dev pgm, developmental programme; Dev S,
developmental skills; Dev stim, developmental stimulation; DPI, developmental programme intervention; E, experimental group;
Hm, home, Hp, hospital; I, instruction; IS, infant stimulation; KC, Kangaroo Care; mo, months (corrected age); NDT, neurodevelopmental
treatment; NIDCAP, Newborn Individualized Developmental Care and Assessment Program; P, parents; P, enhance parental skills;
PIT, parentinfant treatment; PMA, postmenstrual age; Phys T, physical therapy; Sens enr, sensory enrichment; T, parents are therapist; T
stim/RF tactile stimulation/range finding; TT, treadmill training; Wb, waterbed; ?, no information available.

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EFFECT OF INTERVENTION STARTING BETWEEN DISCHARGE FROM according to Vojta.35 It concluded that outcome in the two
NICU AND 9 MONTHS CORRECTED AGE treatment groups did not show a statistically significant dif-
Six of the eight studies in which intervention started in the ference. The other study reported a doseresponse effect of
period between discharge from the NICU and the corrected treatment, according to Vojta, on motor outcome.36
age of 9 months had a high methodological quality. In four
studies, which included the two methodologically strongest EFFECT OF INTERVENTION STARTING BETWEEN 9 AND 18 MONTHS
studies, the effects of NDT were evaluated.2932 None of the CORRECTED AGE
NDT studies demonstrated a statistically significant effect of Three of the six post-NICU studies, in which intervention
intervention on motor development. However, it should be started between the corrected ages of 9 and 18 months,
noted that outcome in two studies was evaluated only by had a high methodological quality. One study demonstrat-
means of global developmental tests.29,31 Two other studies ed a positive effect of specific motor training on motor
applied a programme to stimulate infant motor develop- development of infants with Down syndrome;37 another
ment.33,34 Both programmes consisted of general sensory revealed that a general infant stimulation programme facili-
stimulation and general stimulation of motor development. tated motor development to a greater degree than NDT.10,11
In addition, the intervention in the Barrera et al. study33 The third did not find a significant effect of conductive
included enhancement of parentinfant interaction; that of education on motor development.38 The other three late
Leksculchai and Cole34 included passive handling techniques. post-NICU studies had a more limited methodological
Both studies reported a statistically significant positive effect quality. One evaluated the effect of NDT and did not
of intervention on motor development. Two studies address- demonstrate a significant positive effect of intervention.39
ing the effect of intervention starting in this age period were of The other two used general programmes to stimulate
limited methodological quality. One study evaluated, in motor development. One of them found a beneficial effect
relatively small groups, the effect of NDT and of treatment of intervention.40

Table V: Composition of programmes used in early intervention

Intervention Sensory stimulation Motor intervention Enhancing


Stress Specific Specific General Passive Active Active PII
reduction unimodal multimodal multimodal specific general

Waterbed14,21 + + +
ATVV intervention16 + +
NIDCAP13,15,18,22,23,27,28 + + + +
KC17,19,24 + + + +
Developmental handling26 + + + ?
Treadmill training37 + +
Tactile stimulation/range finding25 + +
Vojta35,36 + +
Sensory enrichment20 +
Developmental intervention8,9 + + + +
NDT10,11,29-31,35,39,4143 + + +
Developmental programme34 + + +
Developmental milestones58 + + +
Developmental stimulation40 + + +
Developmental parent intervention33 + + +
Infant stimulation10,11 + + +
Conductive education38 + +
Developmental skills39 + + ?
Parentinfant treatment33 +
Notes
Stress reduction: decreasing stressful events to body by restricting input from environment until infant is capable of maintaining an adequate
organization of its behavioural state; placing infant in such a way as to provide a sense of containment similar to intrauterine environment.
Sensory stimulation: (a) specific unimodal: procedures during which a single sensory modality is stimulated (e.g. specific tactile
stimulation); (b) specific multimodal: procedures during which multiple specific sensory modalities are stimulated (e.g. ATVV, which consists
of application of auditory, tactile, visual, and vestibular stimuli); (c) general multimodal: procedures during which multiple forms of not
explicitly described sensory stimuli are applied (e.g. verbal and tactile encouragement as part of general developmental programmes).
Motor intervention strategies: (a) passive procedures: therapist or parent performs specific techniques, which do not require active motor
behaviour of child, i.e. child has a passive role (e.g. handling, positioning, and facilitation procedures); (b) active specific: child is encouraged
to actively train a specific motor ability (e.g. walking by means of treadmill training); (c) active general: child is encouraged to train a variety of
motor abilities; stimulation of activities occurs in general by means of structured activities, which are designed to meet childs developmental
level. Practice and play are important elements in this last type of intervention.
Enhancing parentinfant interaction: parents receive information on infant behaviour. Increased knowledge on infantile behaviour facilitates
parents sensitivity to childs needs and promotes developmentally supportive behaviour. +, procedure used; , procedure not used; ?, no
information available; PII, parentinfant interaction. For other abbreviations see legends to Table IV.

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EFFECT OF INTERVENTION STARTING BETWEEN NICU DISCHARGE Discussion
AND 18 MONTHS CORRECTED AGE We are not the first to write a review on the effects of EI for
The three studies in which intervention started between children at high risk for developmental disabilities; others
discharge from the NICU and a corrected age of 18 months have preceded us.6,4450 The major conclusion from these
were all level I studies (see Table I) with a moderate to high reviews is that the evidence favouring EI is inconclusive.
internal validity and a moderate external validity. Two studies Results from this present review indicate that we have moved
evaluated the effect of NDT. The study that assessed the effect a little way forwards. In the following sections we shall point
of a short period of intensive NDT was unable to demonstrate out the direction of progress. However, before we address the
a significant effect of intervention on motor development;41 issues of which programme is best applied and at what age,
the other, which applied less intensive NDT for half a year, we first discuss some methodological issues.
reported a positive effect of intervention on motor develop-
ment.42 The third study evaluated the effect of a general physi- METHODOLOGICAL CONSIDERATIONS
cal therapy programme and did not find a significant beneficial The studies included in this review were very heterogeneous
effect of the intervention on motor development.43 in nature. A large variation existed not only in the number of

Table VI: Evaluation of effect of intervention

Study Ages of evaluation Outcome measuresa Resultsb


Preterm Term 19mo 918mo >18mo Neuromotor Developmental

NICU
Als et al.13 1 9 APIB, Bayley PDI Bayley MDI E>C
Resnick et al.8,9 12 24 Bayley PDI Bayley MDI E>C
Darrah et al.14 1 + 4,8 12 18 MAI Peabody E=C
Ariagno et al.15 1 1,4 12 24 Bayley PDI, NAPI, APIB Bayley MDI E=C
Charpak et al.17 + 3,6 9, 12 Griffiths E=C
Nelson et al.16 2,4 12 Bayley PDI Bayley MDI E=C
Kleberg et al.18 12 Bayley PDI Bayley MDI E=C
Tessier et al.19 12 Griffiths E=C
Westrup et al.28 66 Movement ABC E=C
Korner et al.21 1 LAPPI E>C
Feldman et al. 24 1 3,6 Bayley PDI Bayley MDI E>C
Helders et al.25 Pre, after PM Dev E=C
2wk, weekly
till discharge
Als et al.22 4 + 1,3,6 9 APIB, Bayley PDI Bayley MDI E>C
Leib et al.20 2 6 Bayley PDI, NBAS Bayley MDI E>C
Mouradian and Als23 1 APIB E>C
Becker et al.26 3 VA E=C
Kleberg et al.27 36 Griffiths E=C
After NICU to 9mo
Goodman et al.29 6 9, 12 Griffiths E=C
Piper et al.30 6 12 Wolanski Milani, Griffiths E=C
Leksculchai and Cole34 + 1,2,3,4 TIMP E>C
Barrera et al.33 4 16 Bayley PDI Bayley MDI E>C
Weindling et al.32 12 30 MAI Griffiths E=C
Rothberg et al.31 72 GriffithsII E=C
dAvignon et al.35 3672 CP class E=C
Kanda et al.36 1,2,3,5 Every 3 mo 59 Clin Neur Ex E>C
918mo
Ulrich et al.37 Every 2 wk Bayley PDI Bayley MDI E>C
Palmer et al.10,11 18,24 Bayley PDI Bayley MDI E<C
Reddihough et al.38 Prepost GMFM E=C
Mahoney39 14, 26 Bayley MDI, Peabody E=C
Eickmann et al.40 12 18 Bayley PDI Bayley MDI E>C
Piper and Pless58 Prepost Griffith E=C
After NICU to 18mo
Mayo42 Pre Post Wolanski Bayley MDI, Gesell E>C
Scherzer et al.43 Pre 24 MDC Gesell E=C
Harris41 Pre Post Bayley PDI Bayley MDI, Peabody E=C
aFor explanation of abbreviations see Table VII. bStatistically significant differences found in motor outcome at oldest age of evaluation:

E>C, experimental group significantly better outcome than control group; E=C, no difference between groups; E<C, control group better
outcome than experimental group.E, experimental group; C, control group; Pre, before intervention; Post, after intervention; +, present;
, absent.

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participants included in the studies but also in the interven- problem concerns the existing standardized outcome mea-
tion methods that started before 18 months. The interventions sures. In general, they are characterized by a lack of sensitiv-
themselves, the outcome measures used to evaluate the inter- ity in detecting small changes in motor development,
vention, and the ages at which outcome was determined were although these small changes might have an important influ-
too heterogeneous to permit a formal meta-analysis. The ence on the functional abilities of the child. Not only did the
methods of intervention in the NICU period varied from studies included in this review use a large variation of out-
NIDCAP and Kangaroo Care to different kinds of stimulation come measures (see Table VII), but the measures were also
programmes. In post-NICU studies NDT was the leading mainly discriminative. The use of discriminative measures,
method of intervention, but there were also studies that used which focus on the comparison of a childs score with an
interventions such as infant stimulation, conductive educa- age-equivalent score, might be one of the reasons that so lit-
tion, and other developmental programmes. tle effect of EI is found. Another problem associated with the
It was encouraging to find that 20 of 34 studies had a high outcome measures used is that they mainly quantify quanti-
methodological quality, i.e. they had an evidence level of I and tative changes in motor development, rather than qualitative
fair to high internal and external validity. This reflects the fact changes and measures that focus on changes in functional
that, during recent years, the requirement of good quality stud- abilities.52
ies on the effect of EI has increasingly been met.51 Previous Few studies addressed the effect of intervention on out-
studies have indicated that with an increase in the rigour of the come beyond preschool age. This means that we lack informa-
studies, the support for effectiveness of EI decreases.45,51 This tion on the effect of EI on the childs activities during daily life
also holds true for the studies included in the present review. and the childs socialization brought about by the potentially
Of the 20 studies with a high methodological quality only six beneficial effect of EI on motor development. Future studies
(30%) were able to demonstrate a significant beneficial effect of should address the effect of EI on these outcome parameters,
intervention on motor development. Of the 14 studies with a because they have a major impact on the childs participation
limited methodological quality, seven (50%) reported a posi- in society.
tive effect of intervention. Results of our review are discussed with age at onset of inter-
Studies on the effect of intervention in children with or at vention as a primary focus. The number of studies with a high
risk for developmental disorders like the studies included in methodological quality in the various age periods after NICU
the present review are often hampered by specific prob- discharge was so small that it precluded conclusions on the
lems.50,51 First, many studies include small study groups with a effect of age at onset of intervention after term age. Therefore,
large heterogeneity of degree and type of problems, thereby we discuss the results of our review in two sections, one for
diminishing generalizability and statistical power. Second, NICU studies and one for post-NICU studies.
assigning participants to a control group that does not receive
treatment is usually considered unethical. Results of most NICU STUDIES
intervention studies, therefore, represent only the additional Eight of the 17 NICU studies had a high methodological quality;
value of the intervention under study. Another significant three of those evaluated the effects of NIDCAP intervention.

Table VII: Classification of outcome measures into neuromotor tests and developmental tests

Neuromotor test Abbreviation Dates Developmental test Abbreviation Dates

Bayley Scales of Infant Development BayleyPDI 1969, 1993 Griffiths Developmental Scales Griffiths 1954, 1970
Psychomotor Development Index MilaniComparetti Motor Milani 1967
Development Screening test
Wolanski Gross Motor Evaluation Wolanski 1973 Bayley Scales of Infant Development Bayley MDI 1969, 1993
Neonatal Behavioral Assessment Score NBAS 1973, 1984 Mental Development Index
Motor Development Checklist MDC 1976 Gesell Developmental Schedules Gesell 1974
Movement Assessment of Infants MAI 1980 Peabody Developmental Motor Scales Peabody 1974
Assessment of Preterm Infants Behavior APIB 1982 Muenchener Funktionelle MFED 1978
Longitudinal Neurobehavioral LAPPI 1983 Entwicklungs Diagnostik
Assessment Procedure for Preterm Infants Psychomotor Development profile PM Dev 1981
Supplemental Motor Test for Postural PoCo 1987 (based on Gesell)
Control
Neurobehavioral Assessment of the NAPI 1990
Preterm Infant
Movement Assessment Battery for Movement ABC 1992
Children
Gross Motor Function Measure GMFM 1993
Test of Infant Motor Performance TIMP 1995
Video-analysis (amount and variation VA 1999
in movements)
Clinical neurological examination Clin Neur Ex nfs
CP classification CP class 1981

nfs, not further specified.

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http://dx.doi.org/10.1017/S0012162205000824
One demonstrated a significant positive effect on motor devel- POST- NICU STUDIES
opment, as measured by the Psychomotor Index of the Bayley Of the 17 studies that started after the NICU period, 12 had a
Scales of Infant Development (BayleyPDI13), but the other high methodological quality. Only four of these were able to
two did not reveal such an effect on BayleyPDI.15,18 The differ- show a beneficial effect of intervention on motor develop-
ence in outcome between the three studies might be attributed ment. Eight of the 12 studies evaluated the effects of NDT or
to the age at which outcome was assessed. Outcome in the physiotherapy, mainly on the basis of the principles of NDT.
positive effect study of Als et al.13 was evaluated at 9 months It is striking that only one of these studies reported a better
corrected age, and in the two no effect studies at 12 and 24 motor outcome in the experimental group than in the con-
months. It could be, therefore, that NIDCAP has a temporary trol group.42 The positive effect study of Mayo42 differed
beneficial effect on motor development of infants at high risk from the other studies in being the only one that compared
for developmental disorders. This notion is in line with results intensive NDT treatment (once a week) with less intensive
of two recent meta-analyses which concluded that NIDCAP has NDT (once a month). The other eight studies compared
a temporary beneficial effect on cognitive and motor develop- NDT with infant stimulation10,11 or with a form of standard
ment.53,54 However, it should be kept in mind that only the care that was not defined further. In six of the seven studies,
study by Westrup et al.28 assessed the effect of NIDCAP beyond motor outcome in the NDT group was similar to that of the
the age of 2 years but was unable to demonstrate a significant contrast group. In the seventh study, motor development
positive effect of NIDCAP on developmental outcome at the was worse in children treated in accordance with the princi-
age of 512 years. Nevertheless, considering reports that NIDCAP ples of NDT than in children who received an infant stimula-
intervention in low-risk preterm infants has a significant posi- tion programme.10,11 The above studies indicate that NDT
tive effect on electrophysiological and magnetic resonance during the first years of life does not have a measurable posi-
imaging correlates of brain development at 42 weeks postmen- tive effect on motor development. This is in line with the
strual age,55,56 it is conceivable that NIDCAP might affect com- conclusion of a recent review on the effects of NDT for peo-
plex motor behaviour and cognitive abilities at school age. It ple with CP, aged 5 months to 22 years, which stated that
might be that this putatively positive effect will be found in par- NDT did not have a clear beneficial effect on developmental
ticular in low-risk and not high risk preterm infants. outcome.57
Two other high-quality studies used Kangaroo Care to The other four high-quality studies evaluated the effects of
improve motor outcome.17,19 The application of Kangaroo a developmental programme, treadmill training, or conduc-
Care had no effect on developmental outcome as measured tive education. The two developmental programme stud-
by the Griffiths Developmental Scales at 6 and 12 months ies33,34 and the treadmill training study37 reported a positive
corrected age. Two explanations for this result can be effect of intervention on motor development. The fourth
offered. First, it could be that Kangaroo Care does not affect study compared the effect of conductive education with that
motor development. It is likely that the effect of the relatively of traditional neurodevelopmental programmes. Both types
simple Kangaroo Care is weaker than that of the rather com- of intervention were associated with similar degrees of devel-
plex NIDCAP programme. Second, it is possible that the opmental progress.38
effects are too subtle to be detected by the Griffiths scales. Treatment according to Vojta was evaluated in only two
Two high-quality studies applied intervention strategies studies: dAvignon et al.35 compared, in a small randomized
consisting of procedures to reduce stress in combination with trial with a limited methodological quality, the effect of treat-
specific multimodal sensory stimulation, with or without pas- ment according to Vojta with NDT. They reported that the
sive motor intervention procedures.14,16 Neither study was groups did not differ significantly in developmental outcome.
able to find a positive effect of intervention on motor develop- Kanda et al.,36 who studied the effect of the amount of Vojta
ment at 12 and 18 months corrected age. treatment on developmental outcome, reported a better out-
The last high-quality NICU study showed that developmen- come for the group that received sufficient Vojta training than
tal intervention had a significant positive effect on motor devel- for the group that had received insufficient Vojta therapy.
opment.8,9 In this programme, intervention started in the However, a major drawback of the study is that the design suf-
hospital with vestibular and visual stimulation to promote fered from self-selection of the groups.
development. After discharge, intervention was continued for 2 Thus, the current review indicates that intervention pro-
years by means of a developmental programme in the home sit- grammes in the first postnatal years, according to the prin-
uation. Parents had an important role in performing the inter- ciples of NDT or Vojta, do not have a beneficial effect on
vention programme, which consisted of 400 different motor, motor development in children at high risk for develop-
social and cognitive activities complemented by several parent- mental disorders or in children with CP or Down syn-
ing activities. The positive outcome of the intervention can drome. However, substantial evidence has been provided
probably be attributed more to the duration of the programme which suggests that specific developmental training and
and the continuous involvement of the parents in the develop- general developmental programmes in which parents learn
ment of their children than to the NICU part of the intervention. how to promote infant development can produce a positive
In conclusion, the present review provides little evidence effect on motor development.
that intervention during the NICU period in infants at high
risk for developmental disorders has a beneficial effect on Conclusion
motor development. However, a potential advantageous The present review indicates that intervention in children
effect of NIDCAP on motor development cannot be exclud- at risk of developmental disabilities should be adapted to
ed. We recommend that further studies address the effect of the infants age, i.e. the type of intervention that might be
NIDCAP on developmental outcome at school age in low-risk beneficial for infants at preterm age differs from the type that
and high-risk preterm infants. is effective in infants who have reached at least term age.

430 Developmental Medicine & Child Neurology 2005, 47: 421432


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19. Tessier R, Cristo MB, Velez S, Giron M, Nadeau L, Figueroa de
Acknowledgements
Calume Z, Ruiz-Palez JG, Charpak N. (2003) Kangaroo Mother
We gratefully acknowledge the critical comments of Professor Dr BF
Care: a method for protecting high-risk low-birth-weight and
van der Meulen, Dr VB de Graaf-Peters, and Ms T Dirks on a
premature infants against developmental delay. Infant Behav
previous version of the manuscript. CHB-H was supported
Dev 26: 384397.
financially by the Johanna KinderFonds, Stichting Fonds de Gavere
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and the Graduate School for Behavioural and Cognitive
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17th Annual Congress of the


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Childhood Disability
1922 November 2005, Monaco

Satellite Symposia: 21st November

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